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Post-traumatic inflammatory pseudotumor of the esophagus
Brief Reports
S Marchi, F Costa, M Mumolo, et al.
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BRIEF REPORTS Post-traumatic inflammatory pseudotumor of the esophagus Santino Marchi, MD, Francesco Costa, MD, PhD, Maria Gloria Mumolo, MD, Massimo Bellini, MD, PhD, Eugenio Ciancia, MD, Patrizia Giusti, MD, Giacomo Maltinti, MD
Inflammatory pseudotumors (IPTs) are rare, tumor-like benign lesions that occur in various From the Department of Internal Medicine-Section of Gastroenterology and Institute of Radiology, University of Pisa and Pathology Service, S. Chiara Hospital, Pisa, Italy. Reprint requests: Santino Marchi, MD, Dipartimento di Medicina Interna, S.O. Gastroenterologia, Ospedale S. Chiara, Via Roma, 67, 56122 Pisa, Italy. Copyright © 2001 by the American Society for Gastrointestinal Endoscopy 0016-5107/2001/$35.00 + 0 37/54/116324 doi:10.1067/mge.2001.116324 VOLUME 54, NO. 3, 2001
Peroral insertion techniques of self-expanding metal stents for malignant gastric outlet and duodenal stenosis. Gastrointest Endosc 1996;44:468-71. Wayman I, Bliss R, Richardson DL, Griffin SM. Self-expanding metal stents in the palliation of small bowel stenosis secondary to recurrent gastric cancer. Gastrointest Endosc 1998; 47:286-90. Yates MR III, Morgan DE, Baron TH. Palliation of malignant gastric and small intestinal strictures with self-expandable metal stents. Endoscopy 1998;30:266-72. Nevitt WA, Vida F, Kozarek RA, Traverso LW, Raltz SL. Expandable metallic protheses for malignant obstructions of gastric outlet and proximal small bowel. Gastrointest Endosc 1998;47:271-6. Soetikno RM, Carr-Locke DL. Expandable metal stents for gastric-outlet, duodenal, and small intestinal obstruction. Gastrointest Endosc Clin of N Am 1999;9:447-57. Thumbe VK, Houghton AD, Smith MSH. Duodenal perforation by a Wallstent. Endoscopy 2000;32:495-7. Binkert CA, Jost R, Steiner A, Zollikofer CL. Benign and malignant stenoses of the stomach and duodenum: treatment with self-expandable metallic endoprostheses. Radiology 1996;199:335-8. Soetikno RM, Lichtenstein DR, Vandervoort J, Wong RC, Roston AD, Slivka A, et al. Palliation of malignant gastric obstruction using an endoscopically placed Wallstent. Gastrointest Endosc 1998;47:267-70. Mauro MA, Koehler RE, Baron TH. Advances in gastrointestinal intervention: the treatment of gastroduodenal and colorectal obstructions with metallic stents. Radiology 2000; 215:659-69. Tham TCK, Carr-Locke DL, Vandervoort J, Wong RCK, Lichtenstein DR, Dam JV, et al. Management of occluded biliary Wallstents. Gut 1998;42:703-7. Hoepffner N, Foerster EC, Hogemann B, Domschke W. Longterm experience in Wallstent therapy for malignant choledochal stenosis. Endoscopy 1994;26:597-602. Maetani I, Ukita T, Inoue H, Sato M, Igarashi Y, Sakai Y. Microwave coagulation versus insertion of a second stent for occluded biliary metal stent. Hepato-Gastroenterology. In press.
organs and tissues including the GI tract. Despite features that mimic a neoplasm, IPTs probably represent an inflammatory response to a variety of as yet unknown agents. This is a description of a case of a pseudotumor that arose in the proximal third of the esophagus as an unusual complication of a traumatic mucosal injury, and which was followed by spontaneous and complete resolution. CASE REPORT A 49-year-old woman was referred to the emergency room with a 1-month history of substernal pain and mild dysphagia with solid foods. One year earlier she had accidentally swallowed a herring-bone and experienced mild heartburn that resolved spontaneously. The patient was evaluated clinically and antacids were prescribed. Although the pain gradually decreased, intermittent dysphagia with solid foods persisted. Therefore, 2 months later the patient underwent a barium swallow (Fig. 1) that revealed a noninfiltrating polypoid mass (1.5 × 0.7 cm) at the level of the aortic arch. This finding was confirmed by GASTROINTESTINAL ENDOSCOPY
397
S Marchi, F Costa, M Mumolo, et al.
Brief Reports
Figure 3. Photomicrograph of tumor showing small blood vessels, spindle-shaped cells and polymorphous inflammatory infiltrate composed mostly of lymphocytes (H&E, orig. mag. ×100).
Figure 1. Barium contrast radiograph showing non-infiltrating polypoid mass (1.5 × 1 cm) at level of aortic arch.
Figure 4. Photomicrograph showing immunohistochemical staining for CD 34 which highlights endothelial cells. Spindle cells were negative (diaminobenzidine, orig. mag. ×250).
Figure 2. Endoscopic view of pseudotumor: a broad-based pedunculated polyp in proximal third of esophagus.
eosinophils and mast cells (Fig. 3). The overlying epithelium was flattened but not ulcerated (Fig. 3). The vascular spaces were lined by plump endothelial cells. Although there was active proliferation of spindle cells, atypical mitotic figures were absent (Fig. 3). On immunohistochemical staining, the spindle cells were negative for actin, desmin, S-100 protein (diaminobenzidine), and CD34 (Fig. 4). A diagnosis of inflammatory pseudotumor (IPT) was made and follow-up endoscopy recommended. Two months later the patient was asymptomatic; EGD disclosed no evidence of the polypoid mass, and upper GI radiography confirmed that the lesion had completely disappeared.
DISCUSSION CT and the patient was referred to us for EGD, which demonstrated a broad-based (1.5 × 1 cm) pedunculated polyp covered by macroscopically normal mucosa (Fig. 2). Biopsy specimens disclosed an edematous stroma harboring proliferating small blood vessels, spindle-shaped cells, and a polymorphous inflammatory infiltrate composed mostly of lymphocytes and plasma cells with occasional 398
GASTROINTESTINAL ENDOSCOPY
IPTs are rare benign lesions that were first described in the lung1 and later in other organs and tissues.2-4 In the GI tract, although they occur most commonly in the stomach and the distal ileum, they can develop at any point from the esophagus to the colon.5-8 IPTs are usually found incidentally during VOLUME 54, NO. 3, 2001
Brief Reports
endoscopy, but sometimes they may produce obstructive symptoms or, when ulcerated, bleeding. Despite its tumor-like appearance, there is general agreement that the IPT is an inflammatory mass that probably occurs as an overresponse to mucosal damage because of as yet unknown factors. The lesion has been noted after trauma, surgery,9 infarction,10 a nonsteroidal anti-inflammatory druginduced lesion,11 and as a complication of gastroesophageal reflux,12 but in most patients no specific etiologic factor has been identified.7 IPTs in the esophagus are usually located in the mid or distal segment. At EGD, they usually appear as nodules or circumscribed masses, are rarely pedunculated, and frequently are associated with mucosal ulceration.7 In contrast, the lesion in our patient was situated in the proximal third of the esophagus and was covered with normal mucosa. These features are more typical of the fibrovascular polyp, another type of esophageal lesion whose pathogenesis is uncertain.13,14 Histology, however, was diagnostic in our case because it demonstrated a large number of inflammatory cells (including eosinophils) and numerous reactive blood vessels. Conversely, fibrovascular polyps are composed of mature fibrous tissue with blood vessels lined by nonreactive endothelial cells and contain few inflammatory cells.7 Another atypical feature of our case was the spontaneous disappearance of the lesion. Romeu15 and Korula16 described 2 cases of IPT; in 1 patient it was the result of a complication of Mallory-Weiss syndrome and in the other a result of variceal sclerotherapy. In both cases the IPT spontaneously resolved after clearing of submucosal edema and blood reabsorption. In a third case of esophageal IPT, reported by Staples et al.,12 the lesion regressed with anti-reflux therapy. The outcome of the lesion in our patient supports the hypothesis of an abnormal inflammatory tissue reaction (probably induced by the mucosal injury 1 year earlier). Resection has been advocated, but because of high rates of serious and potentially fatal complications, caution is advisable. In view of the considerable number of cases of spontaneous resolution it is
VOLUME 54, NO. 3, 2001
S Marchi, F Costa, M Mumolo, et al.
our belief that, at least in certain cases, a period of follow-up is also advisable before undertaking invasive procedures. REFERENCES 1. Brunn H. Two interesting benign lung tumors of contradictory histopathology: remarks on the necessity for maintaining chest tumor registry. J Thorac Surg 1939;9:119-31. 2. Day DL, Sane S, Dehner LP. Inflammatory pseudotumor of the mesentery and small intestine. Pediatr Radiol 1986;16:210-5. 3. Antony PP, Telesinghe PV. Inflammatory pseudotumor of the liver. J Clin Pathol 1986;39:761-8. 4. Vujanic GM, Berry PJ, Frank JD. Inflammatory pseudotumor of the kidney with extensive metaplastic bone. Pediatr Pathol 1992;12:557-61. 5. Vanek J. Gastric submucosal granuloma with eosinophilic infiltration. Am J Pathol 1949;25:397-411. 6. Kojimahara K, Mukai M, Yamazaki K, Yamada T, Katayama T, Nakada K, et al. Inflammatory pseudotumor of the stomach: report of a highly infiltrative case with electron microscopic and immunohistochemical studies. Acta Pathol Jpn 1993;43:65-70. 7. Li Volsi VA, Perzin KH. Inflammatory pseudotumors (Inflammatory fibrous polyps) of the esophagus. Am J Dig Dis 1975;20:475-81. 8. Koneman EW, Sawyer KC, Lubchenco AE. Eosinophilic granuloma of the ileum. Arch Surg 1959;78:923-7. 9. Wu JP, Yunis EJ, Fetterman G, Jaeschke WF, Gilbert EF. Inflammatory pseudo-tumor of the abdomen: plasma cells granulomas. J Clin Pathol 1973;26:943-8. 10. Matsubara O, Tan-Liu NS, Kenney RM, Mark EJ. Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fibrous histiocytoma or to plasma cell granuloma in 32 cases. Hum Pathol 1988;19:681-9. 11. Sacca’ N, Rodino’ S, De Medici A, De Siena M, Giglio A. NSAID-induced digestive hemorrhage and esophageal pseudotumor: a case report. Endoscopy 1995;27:632. 12. Staples DC, Knodell RG, Johnson LF. Inflammatory pseudotumor of the esophagus. A complication of gastroesophageal reflux. Gastrointest Endosc 1978;24:175-6. 13. Bematz PE, Smith JL, Ellis FH, Anderson HA. Benign pedunculated intraluminal tumors of the esophagus. J Thorac Surg 1958;35:503-12. 14. Scintu F, D’Alia G, Cabras M, Pisano M, Casula G. Giant fibrovascular polyps of the esophagus. Report of a case and review of the literature. Giorn Ital End Dig 1999; 22:33-7. 15. Romeu J. Pseudotumor in the Mallory-Weiss syndrome. Am J Gastroenterol 1978;70:83-4. 16. Korula J. Pseudotumor of the esophagus: an unusual complication of esophageal variceal sclerotherapy. Am J Gastroenterol 1985;80:954-6.
GASTROINTESTINAL ENDOSCOPY
399
S Marchi, F Costa, M Mumolo, et al.
4.
5.
6.
7.
8. 9.
10.
11.
12.
13.
14.
BRIEF REPORTS Post-traumatic inflammatory pseudotumor of the esophagus Santino Marchi, MD, Francesco Costa, MD, PhD, Maria Gloria Mumolo, MD, Massimo Bellini, MD, PhD, Eugenio Ciancia, MD, Patrizia Giusti, MD, Giacomo Maltinti, MD
Inflammatory pseudotumors (IPTs) are rare, tumor-like benign lesions that occur in various From the Department of Internal Medicine-Section of Gastroenterology and Institute of Radiology, University of Pisa and Pathology Service, S. Chiara Hospital, Pisa, Italy. Reprint requests: Santino Marchi, MD, Dipartimento di Medicina Interna, S.O. Gastroenterologia, Ospedale S. Chiara, Via Roma, 67, 56122 Pisa, Italy. Copyright © 2001 by the American Society for Gastrointestinal Endoscopy 0016-5107/2001/$35.00 + 0 37/54/116324 doi:10.1067/mge.2001.116324 VOLUME 54, NO. 3, 2001
Peroral insertion techniques of self-expanding metal stents for malignant gastric outlet and duodenal stenosis. Gastrointest Endosc 1996;44:468-71. Wayman I, Bliss R, Richardson DL, Griffin SM. Self-expanding metal stents in the palliation of small bowel stenosis secondary to recurrent gastric cancer. Gastrointest Endosc 1998; 47:286-90. Yates MR III, Morgan DE, Baron TH. Palliation of malignant gastric and small intestinal strictures with self-expandable metal stents. Endoscopy 1998;30:266-72. Nevitt WA, Vida F, Kozarek RA, Traverso LW, Raltz SL. Expandable metallic protheses for malignant obstructions of gastric outlet and proximal small bowel. Gastrointest Endosc 1998;47:271-6. Soetikno RM, Carr-Locke DL. Expandable metal stents for gastric-outlet, duodenal, and small intestinal obstruction. Gastrointest Endosc Clin of N Am 1999;9:447-57. Thumbe VK, Houghton AD, Smith MSH. Duodenal perforation by a Wallstent. Endoscopy 2000;32:495-7. Binkert CA, Jost R, Steiner A, Zollikofer CL. Benign and malignant stenoses of the stomach and duodenum: treatment with self-expandable metallic endoprostheses. Radiology 1996;199:335-8. Soetikno RM, Lichtenstein DR, Vandervoort J, Wong RC, Roston AD, Slivka A, et al. Palliation of malignant gastric obstruction using an endoscopically placed Wallstent. Gastrointest Endosc 1998;47:267-70. Mauro MA, Koehler RE, Baron TH. Advances in gastrointestinal intervention: the treatment of gastroduodenal and colorectal obstructions with metallic stents. Radiology 2000; 215:659-69. Tham TCK, Carr-Locke DL, Vandervoort J, Wong RCK, Lichtenstein DR, Dam JV, et al. Management of occluded biliary Wallstents. Gut 1998;42:703-7. Hoepffner N, Foerster EC, Hogemann B, Domschke W. Longterm experience in Wallstent therapy for malignant choledochal stenosis. Endoscopy 1994;26:597-602. Maetani I, Ukita T, Inoue H, Sato M, Igarashi Y, Sakai Y. Microwave coagulation versus insertion of a second stent for occluded biliary metal stent. Hepato-Gastroenterology. In press.
organs and tissues including the GI tract. Despite features that mimic a neoplasm, IPTs probably represent an inflammatory response to a variety of as yet unknown agents. This is a description of a case of a pseudotumor that arose in the proximal third of the esophagus as an unusual complication of a traumatic mucosal injury, and which was followed by spontaneous and complete resolution. CASE REPORT A 49-year-old woman was referred to the emergency room with a 1-month history of substernal pain and mild dysphagia with solid foods. One year earlier she had accidentally swallowed a herring-bone and experienced mild heartburn that resolved spontaneously. The patient was evaluated clinically and antacids were prescribed. Although the pain gradually decreased, intermittent dysphagia with solid foods persisted. Therefore, 2 months later the patient underwent a barium swallow (Fig. 1) that revealed a noninfiltrating polypoid mass (1.5 × 0.7 cm) at the level of the aortic arch. This finding was confirmed by GASTROINTESTINAL ENDOSCOPY
397
S Marchi, F Costa, M Mumolo, et al.
Brief Reports
Figure 3. Photomicrograph of tumor showing small blood vessels, spindle-shaped cells and polymorphous inflammatory infiltrate composed mostly of lymphocytes (H&E, orig. mag. ×100).
Figure 1. Barium contrast radiograph showing non-infiltrating polypoid mass (1.5 × 1 cm) at level of aortic arch.
Figure 4. Photomicrograph showing immunohistochemical staining for CD 34 which highlights endothelial cells. Spindle cells were negative (diaminobenzidine, orig. mag. ×250).
Figure 2. Endoscopic view of pseudotumor: a broad-based pedunculated polyp in proximal third of esophagus.
eosinophils and mast cells (Fig. 3). The overlying epithelium was flattened but not ulcerated (Fig. 3). The vascular spaces were lined by plump endothelial cells. Although there was active proliferation of spindle cells, atypical mitotic figures were absent (Fig. 3). On immunohistochemical staining, the spindle cells were negative for actin, desmin, S-100 protein (diaminobenzidine), and CD34 (Fig. 4). A diagnosis of inflammatory pseudotumor (IPT) was made and follow-up endoscopy recommended. Two months later the patient was asymptomatic; EGD disclosed no evidence of the polypoid mass, and upper GI radiography confirmed that the lesion had completely disappeared.
DISCUSSION CT and the patient was referred to us for EGD, which demonstrated a broad-based (1.5 × 1 cm) pedunculated polyp covered by macroscopically normal mucosa (Fig. 2). Biopsy specimens disclosed an edematous stroma harboring proliferating small blood vessels, spindle-shaped cells, and a polymorphous inflammatory infiltrate composed mostly of lymphocytes and plasma cells with occasional 398
GASTROINTESTINAL ENDOSCOPY
IPTs are rare benign lesions that were first described in the lung1 and later in other organs and tissues.2-4 In the GI tract, although they occur most commonly in the stomach and the distal ileum, they can develop at any point from the esophagus to the colon.5-8 IPTs are usually found incidentally during VOLUME 54, NO. 3, 2001
Brief Reports
endoscopy, but sometimes they may produce obstructive symptoms or, when ulcerated, bleeding. Despite its tumor-like appearance, there is general agreement that the IPT is an inflammatory mass that probably occurs as an overresponse to mucosal damage because of as yet unknown factors. The lesion has been noted after trauma, surgery,9 infarction,10 a nonsteroidal anti-inflammatory druginduced lesion,11 and as a complication of gastroesophageal reflux,12 but in most patients no specific etiologic factor has been identified.7 IPTs in the esophagus are usually located in the mid or distal segment. At EGD, they usually appear as nodules or circumscribed masses, are rarely pedunculated, and frequently are associated with mucosal ulceration.7 In contrast, the lesion in our patient was situated in the proximal third of the esophagus and was covered with normal mucosa. These features are more typical of the fibrovascular polyp, another type of esophageal lesion whose pathogenesis is uncertain.13,14 Histology, however, was diagnostic in our case because it demonstrated a large number of inflammatory cells (including eosinophils) and numerous reactive blood vessels. Conversely, fibrovascular polyps are composed of mature fibrous tissue with blood vessels lined by nonreactive endothelial cells and contain few inflammatory cells.7 Another atypical feature of our case was the spontaneous disappearance of the lesion. Romeu15 and Korula16 described 2 cases of IPT; in 1 patient it was the result of a complication of Mallory-Weiss syndrome and in the other a result of variceal sclerotherapy. In both cases the IPT spontaneously resolved after clearing of submucosal edema and blood reabsorption. In a third case of esophageal IPT, reported by Staples et al.,12 the lesion regressed with anti-reflux therapy. The outcome of the lesion in our patient supports the hypothesis of an abnormal inflammatory tissue reaction (probably induced by the mucosal injury 1 year earlier). Resection has been advocated, but because of high rates of serious and potentially fatal complications, caution is advisable. In view of the considerable number of cases of spontaneous resolution it is
VOLUME 54, NO. 3, 2001
S Marchi, F Costa, M Mumolo, et al.
our belief that, at least in certain cases, a period of follow-up is also advisable before undertaking invasive procedures. REFERENCES 1. Brunn H. Two interesting benign lung tumors of contradictory histopathology: remarks on the necessity for maintaining chest tumor registry. J Thorac Surg 1939;9:119-31. 2. Day DL, Sane S, Dehner LP. Inflammatory pseudotumor of the mesentery and small intestine. Pediatr Radiol 1986;16:210-5. 3. Antony PP, Telesinghe PV. Inflammatory pseudotumor of the liver. J Clin Pathol 1986;39:761-8. 4. Vujanic GM, Berry PJ, Frank JD. Inflammatory pseudotumor of the kidney with extensive metaplastic bone. Pediatr Pathol 1992;12:557-61. 5. Vanek J. Gastric submucosal granuloma with eosinophilic infiltration. Am J Pathol 1949;25:397-411. 6. Kojimahara K, Mukai M, Yamazaki K, Yamada T, Katayama T, Nakada K, et al. Inflammatory pseudotumor of the stomach: report of a highly infiltrative case with electron microscopic and immunohistochemical studies. Acta Pathol Jpn 1993;43:65-70. 7. Li Volsi VA, Perzin KH. Inflammatory pseudotumors (Inflammatory fibrous polyps) of the esophagus. Am J Dig Dis 1975;20:475-81. 8. Koneman EW, Sawyer KC, Lubchenco AE. Eosinophilic granuloma of the ileum. Arch Surg 1959;78:923-7. 9. Wu JP, Yunis EJ, Fetterman G, Jaeschke WF, Gilbert EF. Inflammatory pseudo-tumor of the abdomen: plasma cells granulomas. J Clin Pathol 1973;26:943-8. 10. Matsubara O, Tan-Liu NS, Kenney RM, Mark EJ. Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fibrous histiocytoma or to plasma cell granuloma in 32 cases. Hum Pathol 1988;19:681-9. 11. Sacca’ N, Rodino’ S, De Medici A, De Siena M, Giglio A. NSAID-induced digestive hemorrhage and esophageal pseudotumor: a case report. Endoscopy 1995;27:632. 12. Staples DC, Knodell RG, Johnson LF. Inflammatory pseudotumor of the esophagus. A complication of gastroesophageal reflux. Gastrointest Endosc 1978;24:175-6. 13. Bematz PE, Smith JL, Ellis FH, Anderson HA. Benign pedunculated intraluminal tumors of the esophagus. J Thorac Surg 1958;35:503-12. 14. Scintu F, D’Alia G, Cabras M, Pisano M, Casula G. Giant fibrovascular polyps of the esophagus. Report of a case and review of the literature. Giorn Ital End Dig 1999; 22:33-7. 15. Romeu J. Pseudotumor in the Mallory-Weiss syndrome. Am J Gastroenterol 1978;70:83-4. 16. Korula J. Pseudotumor of the esophagus: an unusual complication of esophageal variceal sclerotherapy. Am J Gastroenterol 1985;80:954-6.
GASTROINTESTINAL ENDOSCOPY
399